Rare Presentation of Chorioadenoma Destruens as Acute Haemoperitoneum Mimicking Ruptured Ectopic Pregnancy
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KATHMANDU UNIVERSITY MEDICAL JOURNAL Rare Presentation of Chorioadenoma Destruens as Acute Haemoperitoneum Mimicking Ruptured Ectopic Pregnancy Sinha M,1 Kaur R,1 Gupta R,2 Rani R,1 Aggarwal A1 Department of Obstetrics & Gynaecology 1 Kasturba Hospital, Delhi, India ABSTRACT West Virginia University- Charleston Division 2 Gestational trophoblastic neoplasms (GTN) are proliferative degenerative disorders Charleston Area Medical Center of placental elements and include complete or partial mole (90%), invasivemole (5-8%), choriocarcinoma (1-2%) and placental site tumor (1-2%). Chorioadenoma Charleston, West Virginia, USA destruens is a trophoblastic tumor, characterized by myometrial invasion through direct extension or via venous channels. We present a case of invasive mole eroding uterus and uterine vasculature, causing sudden rupture of uterus with massive Corresponding Author haemoperitoneum mimicking ectopic pregnancy. A 20 year old G1P0 at 6 weeks Ramandeep Kaur gestation presented in Casualty of Kasturba Hospital complaining of severe acute onset lower abdominal pain for one hour. Clinical examination revealed shock. Department of Obstetrics & Gynaecology Sonography suggested ectopic pregnancy and immediate exploratory laparotomy Kasturba Hospital, Delhi, India was decided. On laparotomy, 2000cc of haemoperitoneum was noted. Grape like vesicles protruding through fundal perforation with profuse active bleeding was E-mail: ramandeepkh@yahoo.in seen. Bleeding persisted despite evacuation. Step wise uterine devascularisation failed to achieve haemostasis. Total abdominal hysterectomy was performed as a life saving measure. Citation Sinha M, Kaur R, Gupta R, Rani R, Aggarwal A. Rare Presentation of Chorioadenoma Destruens as Acute KEY WORDS Haemoperitoneum Mimicking Ruptured Ectopic Pregnancy. Kathmandu Univ Med J 2014;48(4):288-91. Ectopic pregnancy, haemoperitonem, invasive mole INTRODUCTION Invasive mole or chorioadenoma destruens comprises 15 chorioadenoma destruens may present rarely as ruptured % of all gestational trophoblastic neoplasia (GTN).1 GTN ectopic pregnancy in shock, incomplete abortion, is characterized by histological abnormalities of chorionic menorraghia or carcinoma endometrium.6,7 villi with oedema of villous stroma and varying degrees of We present a case of invasive mole which gradually eroded trophoblastic proliferation. Absence or presence of fetal or in the uterus and the uterine vasculature, leading to embryonic elements classifies complete or partial mole. sudden rupture of uterus, massive hemoperitoneum and Molar pregnancies which fail to regress result in invasive shock, mimicking ectopic pregnancy. mole, choriocarcinoma, persistent trophoblastic tumor or placental site trophoblastic tumor.2 Molar pregnancies become Invasive moles in 20% CASE REPORT and are commoner in complete molar pregnancies.3,4 A 20 year old G1P0 at 6 weeks gestational age presented Chorioadenoma destruens is a trophoblastic tumor, to the emergency room of Kasturba Hospital complaining characterized by myometrial invasion through direct of severe lower abdominal pain, acute in onset and for the extension or via venous channels with persistence of past one hour. Her pain was severe, colicky in nature and edematous chorionic villi and trophoblastic proliferation radiated to the back and thighs. She denied any nausea, invading myometrium.5 The presence of villi in the vomiting or fever. Her past medical and surgical histories trophoblastic tissue differentiates an invasive mole were unremarkable. She denied any known drug allergies from choriocarcinoma Instead of vaginal bleeding (97%) and was not on any medications. She attained menarche at Page 288
Case Note VOL. 12 | NO. 4 | ISSUE 48 | OCT- DEC 2014 Figure 1. Ultra Sonography prior to surgery Figure 2. Uterine fundal perforation showing Figure 3. Uterine fundal perforation grape like vesicles protruding through the showing grape like vesicles protruding perforation through the perforation& profuse bleeding Figure 4. During hysterectomy- uterus showing Figure 5. Cut section of the uterus following hysterectomy large perforation showing chorioadenoma destruens with tissue & Blood clot the age of 12 years and her menstrual cycles were regular. On, exploratory laparotomy, 2000cc of hemoperitoneum She denied any pregnancies in the past and had a marriage was noted, with peritoneal cavity full of blood and clots. history of one year. Uterus was perforated at the fundus and profuse active bleeding was noted at the site of perforation. Grape like On examination patient was awake, alert and oriented but vesicles were protruding through the uterine perforation was in apparent distress. She was hypotensive with BP of which measure approximately 3 cm x 3 cm (Fig 2, Fig 3). The 90/60 mm of Hg, tachycardic with pulse of 112 b/min. She uterus was eight weeks in size, soft and enlarged, bilateral was pale clinically, afebrile and her respiratory rate was adenxae were normal. On further abdominal exploration, 16/min. Her cardiac, respiratory and neurological system no ectopic tissue or metastatic tissue was identified. evaluation was unremarkable Profuse ongoing bleeding was seen from site of perforation On abdominal examination, she was severely tender in following complete evacuation of the uterus and patient bilateral lower quadrants, with presence of guarding, continued to remain in a state of shock despite adequate rigidity and rebound tenderness. On bimanual examination, fluid replacement, 4 units of blood transfusion and 20 units cervical os was closed, the uterine size could not be assessed of continuous oxytocin infusion. To achieve haemostasis, due to presence of guarding and tenderness. These step wise systematic uterine devascularization was findings were highly suggestive of ectopic pregnancy and attempted but complete haemostasis could be not be emergency transvaginal ultrasound (TVS) was performed, achieved and patient continued to bleed profusely from which demonstrated anteverted uterus, measuring the perforated site. Decision was made to proceed with approximately – 46 x 60 x 76 mm with irregular echogenic total abdominal hysterectomy as a life saving measure. products seen in uterine cavity. Pouch of Douglas showed Post surgery patient became haemodynamically stable. moderate amount of free fluid. However, a definitive Hysterectomized uterus was then sent for histopathological diagnosis of ectopic pregnancy could not be established. examination (Fig 4, Fig 5). During TVS, patient became drowsy and slipped into a semi Her postoperative course was uncomplicated. comatose condition. Her vitals deteriorated with pulse of 140/min and systolic blood pressure of 60 mm of Hg. On postoperative day one, her vitals improved and her Patient was immediately rushed to operation theatre for hemoglobin was 8 gm/dl. She received additional two units Emergency laparotomy with a definitive suspicion of a of blood transfusion and made an uneventful recovery. ruptured ectopic pregnancy. On laboratory investigation, The postoperative metastatic work-up included her haemoglobin and hematocrit was 6 gram and 20 mg serum biochemistry, chest x-ray and upper abdominal percent, random blood sugar values of 130 mg/dl, Serum Ultrasonography and no evidence of metastasis was Bilirubin 1.2 mg/dl, and Serum creatinine 0.9 mg/dl. Her detected. In view of low levels of serum ß-hCG, patient beta HCG could not be sent as emergency laboratory was kept under surveillance with serial quantitative ß-hCG facilities of Kasturba Hospital does not include this estimation to consider chemotherapy if ß-hCG levels investigation. Page 289
KATHMANDU UNIVERSITY MEDICAL JOURNAL Figure 6. Section from uterus showing a Figure 7. The tumour cells are showing Figure 8. Syncytiotrophoblasts and tumour arising from endometrial surface and vascular invasion. (H&E, X 20) intermediate trophoblasts are infiltrating composed of intermediate trophoblasts and the myometrium as single cell infiltrate. afew multinucleated syncytiotrophoblasts, (H&E, X 20) arranged in sheets. (H&E, X40) remains at a plateau or rises. Postoperatively ß-hCG level of gestational trophoblastic neoplasia with uterine rupture was 110 mIU/ml. Patient was discharged home in stable are often catastrophic owing to profuse bleeding, which condition on postoperative day 3. Her ß-hCG level was could potentially be lethal.
Case Note VOL. 12 | NO. 4 | ISSUE 48 | OCT- DEC 2014 is either unavailable or not affordable and chances of losing Use of chemotherapy in the management of invasive mole is the woman to follow-up are high. The recommendations still debatable, with the evidence of spontaneous regression for post-molar follow-up include serum βhCG levels every of metastatic mole in the literature. Chemoprophylaxis may 1–2 weeks after evacuation until normal, hCG levels 2–4 be particularly useful in patients with a high-risk complete weeks after the first normal level, and hCG surveillance mole when hormonal follow-up is either unavailable or not every 1–2 months for 6 months after the first normal hCG affordable and chances of losing the woman to follow-up level. Serial hCG monitoring of women with disease in are high. We did not consider chemotherapy in our case in prolonged remission (greater than 1 year) often provides view of no evidence of metastasis and low ß-HCG levels on early evidence of recurrence, though GTT may rarely be subsequent follow up. The potential risk of patient being associated with undetectable levels of hCG.8 lost to follow up was very much there but did not happen in our case. In our patient, we were unable to achieve adequate haemostasis by step wise uterine devasculaization attempted with uterine and ovarian vessel ligation. CONCLUSION Unfortunately the surgical emergency team was not well versed with the technique of Internal Iliac Ligation, hence it Chorioadenoma destruen has a potential for myometrial was not attempted and total abdominal hysterectomy was and vascular invasion, leading to uterine perforation and massive hemorrhage. Therefore, to avoid adverse then performed. Although the development of effective consequences it is necessary to identify such cases by early chemotherapy has resulted in improved survival of patients first trimester ultrasound. Continued reporting of these with gestational trophoblastic tumor, hysterectomy cases are important so that the obstetricians are aware remains an important adjuncts in the treatment of a about the possibility of ruptured invasive mole and it should selected subset of patients. be kept as a differential diagnosis in all the young pregnant women presents with acute onset lower abdominal pain. REFERENCES 1. Kumar S, Vimla N, Mittal S. Invasive mole presenting as acute 8. Miller FM, Laing FC. Gestational trophoblatic disease http:// hemoperitoneum. J K science. 2004;6: 159-160. brighamrad. havard.edu/cases/bwh/hcache/34/full.html 2. Berkowitz RS, Goldstein DP, In: Berck JS. Gestational trophoblastic 9. Mackenzie F, Mathers A, Kennedy, J Invasive hydatidiform mole neoplasm. PhiladelphiaLipincott, Williams and Wilkins, 2002.1353- presenting as an acute primary haemoperitoneum. Br J Obstet 74. Gynecol. 1993; 100: 953-54. 3. D. Berek & Novak’s Gynecology, 14th ed, Philadelphia, Lippincott 10. Wilson RB, Hunter IS, Dockerty MB. Chorioadenoma Destruens. Am J Williams & Wilkins, 2007. p1581-1604. Obstet Gynecol. 1961; 81: 546-59. 4. Kittur S, Venktesh, Ramlingappa A. A rare case of invasive mole with 11. Hol K, Junnare K, Shekhawat GS, Damle H. Case of Invasive Mole with silent uterine perforation. Int J. Reprod Contracept Obstet Gynecol Uterine Rupture Presenting as a Hemoperitoneum. Global Research 2013;2:109-10. Analysis, 2013 Dec; 2(12): 195-96. 5. Atala C, Riedemann R, Biotti M, Ramírez F, Paublo M. Invasive mole 12. Branka N, Jelena L. Invasive mole case report of massive uterine with uterine rupture. Rev Chil Obstet Ginecol. 1992;57(5):356-8. destruction- Med Arh. 2008;62:242–243. 6. Singh A and, Ratnani RJ. Heterogenous presentation of Chorioadenoma 13. Mitani Y, Jimi S, Takao N. Partial resection of the uterus for destruens. J Obstet Gynaecol India. Dec 2012; 62(Suppl 1): 71–74. chorioadenoma destruens. J Jpn Obstet Gynecol Soc. 1968 Jul;15(3):163-7. 7. Mandal D, Nandi N, Dey RP, Biswas RR, Bhattacharya AK, Biswas SC. Partial Invasive Molar Pregnancy – Two Case Reports. Al Ameen J Med 14. Goldstein DP and Berkowitz RS. Gestational Trophoblastic Neoplasia. Sci. 2010; 3(1): 91-93 WB Saunders, Philadelphia; 1982. 98-121. Page 291
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